Hyperthermia, not uncommon in severe brainstem stroke, is frequently difficult to control and associated with a poor prognosis and high mortality. Successful treatment of central hyperthermia in patients with brainstem infarction by baclofen has not been described. Following basilar artery occlusion, a 68-year-old female developed prolonged hyperthermia. Her hyperthermia did not respond to any antipyretic treatments. A water-cooling blanket was utilized to control her hyperthermia; however, body temperatures fluctuated at a range of 35.5-40.0 degrees C. After given baclofen 30 mg/day, her body temperatures returned to a normal range and remained stable. Hyperthermia following severe brainstem infarction without any sign of infection or inflammatory sources usually comes from a central origin and is likely associated with dysfunctions of the thermoregulatory system. Baclofen may be one of the treatment choices in patients with neurogenic central hyperthermia.